Case report: A 25-year-old woman with encephalopathy and sensorineural hearing loss was referred to us in order to confirm Susac syndrome, due to bilateral visual loss. Funduscopic examination revealed peripheral retinal microangiopathy.
Discussion: Susac syndrome is typically characterized by the clinical triad of encephalopathy, branch retinal artery occlusions, and hearing loss. In our case, ophthalmic findings were atypical. Interrupted vessels and microaneurisms accumulation in the peripheral retina of both eyes were observed.